Behavioral analysis through the lifespan of mutant zebrafish identifies defects in sensorimotor transformation.

is a genetic risk factor for multiple psychiatric disorders. Compared to the dozens of murine models, there is a paucity of zebrafish models-an organism amenable to high-throughput experimentation. We conducted the longitudinal neurobehavioral analysis of mutant zebrafish across key stages of life. During early developmental stages, mutants exhibited abrogated behavioral responses to sensory stimuli across multiple testing platforms. Moreover, during exposure to an acoustic sensory stimulus, loss of resulted in the abnormal activation of neurons in the pallium, cerebellum, and tectum-anatomical sites involved in the integration of sensory perception and motor control. In adulthood, mutants exhibited sexually dimorphic reduction in anxiogenic behavior in novel paradigms. Together, these findings implicate in sensorimotor processes and the genesis of anxiogenic behaviors, which could be exploited for the development of novel treatments in addition to investigating the biology of sensorimotor transformation in the context of deletion.